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Objective Centrosome/Spindle Pole associated Protein 1 (CSPP1, JBTS21) mutations cause Joubert syndrome (JBTS) and JBTS-related ciliopathies. The large protein isoform CSPP-L is a ciliary protein required for ciliogenesis and stabilization of the ciliopathy protein RPGRIP1L (NPHP8/JBTS7/MKS5/FTM) at the ciliary transition zone (TZ). However, RPGRIP1L is dispensable for ciliogenesis and the mechanism by which CSPP-L promotes ciliogenesis is unclear.

CSPP-L and EB3 localize to centriolar satellites and are required for satellite-dependent recruitment of ciliopathy proteins to the centrosome